Journal
WORLD NEUROSURGERY
Volume 146, Issue -, Pages E1287-E1292Publisher
ELSEVIER SCIENCE INC
DOI: 10.1016/j.wneu.2020.11.153
Keywords
Atlantoaxial instability; Cervical dystonia; Cord compression; Craniovertebral junction; Os odontoideum
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This study presented a case series of 4 patients with longstanding cervical dystonia (CD) and complications of myelopathy or radiculopathy. It found that early-onset CD may lead to cord complications at a young age, with satisfactory treatment outcomes in only a few patients.
OBJECTIVE: Atlantoaxial instability, although rarely reported in the literature, can be associated with cervical dystonia (CD) and may lead to compression of the cord at the craniovertebral junction. We present a case series of 4 patients of longstanding CD with neurologic complications. Treatment strategies and challenges are discussed. METHODS: Retrospective analysis of 4 cases of longstanding CD with complications of myelopathy or radiculopathy. RESULTS: The average age at onset of complications was 28 years (range, 17-37). The average duration of CD was 23.75 years. Narrowing of the craniovertebral junction was seen in 3 patients, of which 2 had os odontoideum, and 1 had rotational malalignment at the atlantoaxial joint. One patient had disc desiccation with bulge and intramedullary signal changes in the cord at C3-4 level. Medical treatment was not satisfactory, but botulinum toxin was partly useful in all. One patient had sequelae of myelopathy and did recover partially after deep brain stimulation. Of the 2 patients who underwent surgical fixation with a fusion of the spine, one improved, and the other had no improvement due to irreversible cord damage. The overall outcome was satisfactory only in 2 patients. CONCLUSIONS: Early-onset CD can lead to cord complications at a young age and at higher levels of the cervical spine and at the cervicovertebral junction. Comprehensive management by a multidisciplinary team is crucial to prevent complications early.
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